A systematic literature
review published between 1990 and 2006 using a qualitative approach was conducted
to explore family members' perception of anorexia and bulimia nervosa patients.
Articles were critically reviewed and a meta-synthesis analysis was carried
out based on a meta-ethnographic method to analyze and summarize data. Of a
total of 3,415 studies, nine met the study inclusion and exclusion criteria.
Reciprocal translation was used for data interpretation allowing to identifying
two concepts: disease awareness and disease impacts. Feelings of impotence were
often described in family reorganization. The study results point to distortions
in the concept of disease associated with family involvement, resulting in changes
in communication, attitudes, and behaviors in a context of impotence.

Recently there
have been an impressive growing number of studies linking family functioning
to different medical conditions. Eisler et al10 have described the
family as a health system for its members and provides an explanatory health-disease
model. Family members act by a set of values, beliefs, knowledge, and practices
for health promotion and disease prevention and treatment. This system comprises
also a model of care, where the family supervises the health status of its members,
makes decisions on the paths to be followed, and constantly follows up and evaluates
their health and disease. According to the World Health Organization23
(WHO), health systems are increasingly requiring that families play the role
of caregiver.

However, the perception
of the family of patients with eating disorders has not been much discussed
in the literature. The few studies on that have found that the impact on the
family's health would be comparable to that of caring for a psychotic patient.27,33

Some qualitative
studies have exposed family functioning from the perspective of patients with
eating disorders.7,29 They identified characteristic patterns of
family interactions apparently associated to the development and maintenance
of eating disorders. It was observed an entanglement, i.e., family members become
mixed together and personal limits are blurred, corresponding to the idea of
inexistence of individual identity separated from the core family. These families
are typically overprotective, controlling, and can hardly manage the separation
and independence of its members. Also, continued importance given to physical
appearance in the family seems to result in a distorted perception of the person
with an eating condition.1-4,20,27

Researches about
the family's perspective are scarce and they are mostly epidemiological studies.,6,15,32
Qualitative investigations are rarely seen in the literature,2,14
and they are commonly based on theoretical references such as the systemic one,
where the disease is part of and have a role in the family structure; and the
psychodynamic one, focused on understanding psychic dynamics. From a systemic
perspective, Perkins et al25 conducted semi-structured interviews
addressing the experience of 20 family members of bulimic patients. Their comprehension
of eating disorder largely involved a mixed perception and underestimation of
disease and feelings of impotence, sadness, and guilt prevailed. Honey &
Halse,14 based on a psychodynamic perspective, carried out in-depth
interviews with 16 mothers and eight fathers of anorexic inpatients. Their coping
strategies were predominantly self-distraction and optimism. These studies have
indicated some distortions perceived by caregivers such as disease conception,
treatment, coping strategies, among others.

The objective of
the present study was to carry out a systematic review on how family members
perceive anorexia and bulimia nervosa patients.

A comprehensive
electronic search of the databases PubMed, Lilacs, SciELO, ISI - Web of Science,
PsycoInfo and EmbaseIt was conducted.

The following keywords
were used for searching databases: one of the keywords of descriptors "eating
disorder," "anorexia nervosa," and "bulimia nervosa" combined to at least one
keyword "qualitative research," "qualitative study," "phenomenology," "perspective,"
"perception," "experiences," "family," and "caregivers" as well as their equivalents
in other languages.

For the selection
of articles, inclusion criteria were qualitative studies reporting the experiences
of family members of patients with eating disorders published in Portuguese,
English, Spanish or French between 1990 and 2006. The studies should clearly
have the following design features: 1- original study including a sample of
both male and female subjects; 2- methodological design and theoretical approach
for data collection and analysis; 3- intentional samples with specific selection
criterion and sample size defined by content saturation; 4- samples including
both adolescents and adults; 5 - detailed description of data analysis such
as procedures for data extraction, number of evaluators, category controls and
use of triangulation.

There were excluded
chapters of books, theses and Master's dissertations as well as studies whose
main purpose was to investigate psychiatric disorders other than eating disorders
or studies that did not explore the family's perspective.

Studies were critically
reviewed based on the Critical Appraisal Skills Program8 (CASP),
a checklist that sets out guidelines for the assessment of quality of qualitative
studies. CASP consists of ten items that allow to categorizing articles based
on their methodological structure. The studies were categorized into A and B.

Category A included
studies with low risk of bias that met at least nine out of ten items proposed:
1- clear and rational objective; 2- adequate design to attain the objectives;
3- presentation and discussion of methods; 4- selection of an intentional sample;
5- description of data collection, instruments and saturation process; 6- relationship
between investigator and subject; 7- ethical considerations; 8- dense sound
analysis; 9- presentation and discussion of results focusing on reliability
and use of triangulation; 10- description of contributions and implication of
the knowledge produced in the study and its limitations as well.

Category B included
studies that met at least five out of ten items and partially met the study
criteria with a moderate risk of bias. Case studies were included in this category.

Noblit & Hare22
refer to meta-synthesis as a method of study based on the thorough analysis
of theories, methods, and results found in qualitative studies. The objective
of meta-synthesis is significantly different from that of meta-analysis as it
implies interpretation rather than aggregation of numeric indexes.

At this step a
meta-ethnographic approach was applied to analyze and synthesize data. Articles
were categorized by two independent reviewers. All categories used in the review
and meta-synthesis were obtained from consensus between the two reviewers. A
meta-synthesis was conducted based on the following steps as proposed by Noblit
& Hare:22

Definition of
the area of interest and guiding question of the case studied - perception
of family members of anorexia and bulimia - and search of studies using appropriate
strategies.

Selection of
relevant studies to the sample according to inclusion and exclusion criteria.

Rereading of
studies and recording of information looking for keywords, subjects and main
categories.

Establishing
a link between the studies, either by similarity or opposition.

Comparison of
studies between them.

Synthesis of
findings, seeking to associate them and prioritizing the most recurrent results.

Documentation
and dissemination of meta-synthesis results.

The final step
of the study is a synthesis to clarify new meanings by primarily grouping subjects
that are progressively refined, creating a new core consensus.

The selected studies
were read and reviewed using a meta-ethnographic approach. It consists of induction
and interpretation, and includes resignification (translation) of results between
studies, allowing to understanding and transferring ideas, concepts, and metaphors
among different studies. While resignifications enable to make comparisons between
different studies, they preserve the structure of relations between concepts.
Resignification is a two-step process as proposed by Noblit & Hare.22
The first step, known as second-order interpretation, is based entirely on original
results and the synthesis per se is carried out. Relevant contexts and concepts
of each study were recorded for better understanding interpretations. Concepts
can be related either by similarity, so-called reciprocal translation, or by
opposition, when there are discrepancies. The second step, known as third-order
interpretation, goes beyond the meaning of original results, and conceptually
advances with rereading of originally synthesized categories. The categories
used in the review and meta-synthesis were obtained from consensus between the
authors. An article considered a gold-standard due its design was used as a
reference to manage comparisons between different studies.35

RESULTS

There were first
identified 3,415 occurrences. After pre-selection based on the reading of titles
and abstracts of all studies retrieved in the electronic search, there were
selected 58 studies that met the pre-established criteria. They were read entirely
and nine studies met the inclusion criteria. This set of articles was assessed
for quality. Based on quality criteria, four studies were included into the
category A and five into B. The studies selected are listed in the Table,
and characterized by type of eating disorder, country, subjects, disease stage,
and type of treatment, as well as method of data collection and level of quality.
Studies were classified based on three theoretical approaches: psychodynamic
(n=4), systemic (n=3), and phenomenological (n=2).

More studies (six)
addressed family members of anorexia than bulimia patients (three), of which
two were exclusively on bulimia and one included bulimia and anorexia. They
included a wide range of informants such as mothers, fathers, siblings, and
husbands, totaling 118 family members. Mothers accounted for 62% of all informants
(n=73).

Most studies conducted
semi-structured interviews, one of them used a focus group11 and
another one was based on letter writing where emotions were reported as in a
diary.35 All articles were published from 2002, which indicates a
very recent literature production. Most of them (five) were carried out in England.

The studies included
in the sample showed the state of family members after the diagnosis of eating
symptoms. Disease severity varied among the studies. Most involved as informants
parents of outpatients (six) and inpatients (three). No studies were found with
accounts of family members of male patients.

Acknowledgment
of the disease

Acknowledgment
of the disease was based on two themes. The first one (family comprehension
about eating disorders) was based on the following categories: mixed perception
of disease, chronicity, underestimation of the disease, and control strategy.
The second one (perception of treatment) was based on satisfaction with treatment
and dissatisfaction with treatment.

A. Family
comprehension about eating disorders

The family comprehension
about the disease, their emotional adjustment and how they manage this situation
was one of the themes analyzed in the synthesis.

Mixed perception
of the disease - the analysis of themes showed family members had difficulties
understanding the eating disorder. Some did not have a clear perception of the
disorder, its etiopathogenesis and prognosis.25,35,36

"I knew something
was wrong, but I wasn't sure what it was."25

Chronicity - parents
perceived the disease as a chronic condition, expressing pessimism regarding
their child's recovery and cure. This concern was only seen in studies including
family members of anorexic patients, as shown in the discourse below: 14,30,35

"I wanted our
daughter back. And it took me quite a while to realize that this actually is
not possible, that it will never happen. So you have to move forward, leave
your old self behind and be happy with what you've got now."14

"It will take
her years to readjust to normal life, maybe she will never make it."35

Underestimation-
of the disease - underestimation was associated to the belief that their daughter's
behavior changes were a typical and natural attitude of adolescents, as well
as to the lack of knowledge on the disease and a mechanism of denial. 25,35

"For quite a
long time I really didn't realize it was an eating disorder. I thought it would
never happen to me. I wasn't aware of that."25

Control strategy
- the disease was perceived as a way for the patient to gain control over her
own life and over other people's life. 7,30

" (.....) the
only thing she can get control is over her own body because we went through
hard times... it is the way she uses it (.....)"30

B. Perception
of treatment

This category illustrates
how family members perceived treatment. It was divided into satisfaction and
dissatisfaction with treatment.

Satisfaction with
treatment - in some studies treatment was described as facilitator of family
life. Most studies showed that either outpatient or inpatient treatment made
the patient stable and facilitated living together in the family. 30,35,36

"Since my daughter
has been admitted to the hospital I see light at the end of the tunnel."35

"The only time
I can relax a little or have some kind of relief is now while she is at the
eating disorder unit because she gets physically healthier with treatment."35

Dissatisfaction
with treatment - family members of patients revealed unaddressed needs such
as information and practical guidance on how to manage the patient for they
felt impotent and needed to share this experience with other people. 30,35-36

"We need guidance
on how to manage daily situations, this is my main problem now.....if she has
an eating binge, what should I do? Should I try and stop her? Should I try to
talk to her? Or should I distract her...(....)"36

Disease impact

This theme was
structured based on four blocs. The first bloc (feelings evoked by the disease
in the family) was subcategorized as sadness, fear, impotence, guilt, hopelessness,
and ambiguity. The second one (changes to the patient) was subcategorized as
personality changes and low school performance. The third (changes in the family
after disease) was subcategorized as dysfunctional communication, role changes,
and increased closeness. The fourth (coping strategies) was subcategorized as
optimism and good mood, self-distraction, cognitive reconstruction, religiousness,
search for information.

A. Feelings
evoked by the disease in the family

The disease evoked
in the family many feelings, sometimes contradictory ones. Guilt, sadness, and
impotence were the most commonly perceived and expressed feelings by family.
There was a prevailing feeling of impotence in the studies.

"I would say
that...while I'm talking to you, L. missed her childhood. I feel it very deeply,
and now we don't have a happy 13-year-old girl, she is not a girl but someone
who is taken by depression and negative feelings as if she were carrying the
world upon her shoulders..(...)"29

Fear - family members
were taken by a fear of losing their relative with the disease. Fear was evident
in anorexia studies. 16,35

Impotence - studies
reported a subjective state of impotence, where the prevailing perception was
that an appropriate action would not significantly affect the outcome. They
felt lack of control over the situation. Many family members felt the disease
had profoundly affected them, and they felt manipulated and controlled. 7,16,25,30,35

"There is nothing
I can do that will make a difference."35

Guilt - guilt is
stressed in many studies. Parents felt somehow responsible for their daughters
developing an eating disorder, and believed their own eating habits and attitudes
could have influenced their daughter's behavior.11,13,16,29 Feeling
guilty is identified in the following discourses:

"I believe I
actually did something, something that caused her this."13

Hopelessness -
several studies reported hopelessness especially regarding with the future.
They saw no alternatives nor could not see any personal choices available and
felt unable to channel their energies to their best interest.11,13,16,18

Ambiguity - some
studies reported a feeling of ambiguity among family members. Patients with
eating disorders did not evoke only feelings of compassion and sympathy, but
also anger, rejection and envy particularly among siblings.11,14,16
Etxeberria et al11 study showed siblings sometimes acted as support
and other times they were bothered by the attention given to their sick sister.

"If I envy her?
They do much more for her than for me."11

B. Changes
to the patient

Family members
talked about themselves and at the same time they mentioned the sick relative
and their behavior changes. The most expressive behavior changes were personality
changes and low school performance.

Personality changes
- this change caused a negative impact on relationships. Some aspects mentioned
were the fact that the patient was hiding things form others, that she was no
longer authentic in her relationships. The patient became extremely meticulous
and organized about her eating habits and other general activities. Aggressive
behavior and mood fluctuations were also mentioned.11,29,30

Low school performance
- according to parents, the patients had a very good school performance before
the disease and then some of them even dropped out of school.11

C. Changes
to the family after disease

New family arrangements
were created and the old ones were changed; e.g., triangulation between the
parents and the sick daughter or expelling of the sick one from the family structure.
There were also role changes in the family. The disease became a new member
of the family that would influence and have control over them all.

Dysfunctional communication
- it is defined as a conflictive, incomplete and superficial form of communication.
Family members reported that, because of the eating disorder, the patients would
argue more, communicate less and lie more to cover up for their disease. The
family relationship would improve as they recovered.11,16,25

"We tried to
talk to her at home, but it would always end up in confrontation."25

"Now that she's
gained some weight, the relationship at home is better, we fight less (.....)"11

Role changes -
it is a switch of functions and roles a member can take on in the family in
the event of a disease. Disease was described as a new member of the family.
Former relationships were changed, and patients sometimes became estranged from
their family. Caregivers were overwhelmed as they had to conciliate care of
the sick one and their work duties, in addition to other tasks. 16,25

Increased closeness
between family members - coping with the disease made members come close to
each other and strengthen family ties as seen in some discourses. The patient
would demand a great deal of attention and external control,14,25,29,30
as observed in the following discourse:

"J. and I were
likely closer because of that, I know it may sound odd, but I believe that emotionally
these things happen (......)"25

D. Coping
strategies

Coping strategies
were either problem-centered or emotion-centered. In problem-centered coping
the individual would make efforts to bring up the problem, work out solutions
and make a choice of action. Emotion-centered coping involved an attempt to
manage the emotional impact of stress using mainly defense psychic mechanisms.

Optimism and good
humor - emotion-centered coping strategies helped in some situations to adjust
or minimize the disease impact, but could also indicate distortions and defense
mechanisms.14,25,35

"I'm always
trying to be optimistic (...) you have to think about it positively."25

Self-distraction
- it can involve professional activities, physical exercises, manual work and
leisure activities. This is an emotion-centered coping strategy.14,25,35

"I get immersed
in my work and other projects but then she comes to my mind."35

Cognitive reconstruction
- strategies of cognitive reconstruction consist of efforts by family members
to find positive aspects in the disease experience and seize it as an opportunity
for personal growth by giving it a less threatening and painful meaning. 14,25,35

"It has changed
my life, my opinions and the way I see things, it has been positive in this
sense....(.....)."25

"Everyday I
try to find something positive."14

Religiousness -
comprises accounts of religiousness to cope with the disease based on faith
in recovery or spirituality to find a meaning to the illness. 26,37

Search for more
information - it was noted in some accounts a search for information about the
disease and treatment. Parents sought to get information in textbooks and on
the internet and by talking to health providers. Information about the disease
helps alleviating the feeling of impotence and answering questions, making them
feel more confident and reassured. 14,25,36

"I have been
doing that and it has been very helpful, especially for things I used to find
difficult to do."14

Third-order
interpretation

In the meta-synthesis,
themes of each individual study comprised metacategories that allowed an overall
analysis. The new metacategory "family restructuring" was related to two second-order
groupings: acknowledgment of the disease and disease impact. This metacategory,
which was not individually found in specific studies, included different information
found from the studies.

Eating disorders
lead to overall family restructuring, changing its dynamics, and requiring emotional,
cognitive, and material adjustments. Family life would revolve around the affected
member, leading to changes in the interactions between other family members.
A notable estrangement from other children could be seen. Negative feelings
such as hopelessness and impotence emerged. Adaptative mechanisms such as self-distraction,
optimistic thinking, good humor and religiousness were also seen. It can also
be noted social withdrawal since the disease causes a burden that disrupts the
existing routine.

Eating disorder
is an experience that calls for adaptative challenges of different degrees.
Family restructuring does not necessarily mean resolution, i.e, fully understanding
and accepting the disease, but rather is an organization of less conflictive
ways of coping with it.

DISCUSSION

Family members
have diverse comprehensions as well as a wide variety of viewpoints on anorexia
and bulimia. Studies reported mixed perception of the disease, underestimation
of the problem, understanding of the disease as a chronic condition and as a
strategy of control over one's own life and the life of others.

The first contact
with the disease makes parents anxious and fearful. They quite often express
their disbelief regarding the diagnosis. Parents go through different phases,
the so-called first reactions (shock, disbelief, confusion, fear, anger, conflict,
stress) that can have varying duration, and then a long denial phase, that affect
the whole family, hindering an appropriate adjustment.38

Some of these situations
may be determinant for making the decision whether to seek or not professional
help.

Failure to acknowledge
the disease may delay treatment start and aggravate the condition by making
it more chronic and increasing the risk of death in severe cases.34

The most recurrent
themes found in the studies were impotence, sadness, fear, and guilt. Feelings
of impotence and guilt were recurrent and caused family suffering. Pichon-Rivière,26
while studying family relationships, found that parents tend to believe they
are able to determine what is right and what is wrong to their children. Failure
to do so, indicated by their child's eating disorder, makes them experience
a profusion of feelings such as discomfort, distress, and impotence. They feel
helpless and believe they are bad parents and that they are unable to help.

Family support
groups help easing these feelings of guilt, sadness, helplessness and to express
their heavy emotional burden.

The sick person
suffers as well as their entire family. Before disease onset, the family environment
is described as "normal" and the affected person as healthy. Conflicts were
either inexistent (harmonic family pattern) or they did exist but were not perceived
by the family. Even though when they are perceived in family interactions, it
is hard to associate conflicts to the manifestation or persistence of an eating
disorder.6,21,25

Bruch2
have investigated family functioning before symptom onset and stressed that
there is inadequate interpersonal relationships, characterized by apparent harmony
that covers up severe latent conflicts. Lawrence17 and other authors
have corroborated these findings, pointing out the role of psychological conflicts
in family relationships of patients with eating disorders.

After its onset,
disease manifests as a unit in which family members are affected showing varying
degrees of symptoms. The family's response has also an effect on the patient.
In general, the entire family structure needs restructuring to cope with and
to adjust to the new circumstances of disease and treatment. The contact with
medical procedures (consultations, testing, and several therapies) and sometimes
with hospital settings lead to changes in the daily home routine, disorganization,
and distress. Any intervention should take into account the family environment.
Gabbard13 (1992) proposes family therapy and counseling to parents
as strategies for treating adolescent patients as they are a source of support
and can provide information on the condition. This author believes that addressing
the family allows to address relationship problems that perpetuate the symptom.
Likewise, Eisler et al10 have conducted a randomized clinical trial
to assess the efficacy of two different family interventions compared to a control.
One intervention was applied to the patient, and the other one was a support
to the patient and involved family counseling. The results showed significant
symptomatic and psychological improvement of individual and family functions
in both approaches. Dare et al,9 in a randomized clinical trial with
adult patients, reported the results of a comparison among two psychodynamic
treatments (focal psychodynamic psychotherapy and cognitive-analytic therapy),
family therapy, and control. No statistical difference was observed between
the different psychotherapy approaches, but focal psychotherapy and family therapy
proved to be more effective for gaining weight when compared to the control
treatment.

Studies on coping
mechanisms for eating disorders show that family members use more emotion-centered
strategies to protect themselves and keep a certain level of stability in family
life. But these strategies may make family members neglect the patient's actual
health status.

Folkman et al12
propose that the term coping should be used to express skills that help manage
and adjust to stress situations. According to Carver & Scheier,5
coping is a response to stress to reduce its adverse effects. These strategies
address specific demands arising from crisis and are a burden to one's personal
resources. They can be learned, but people should be advised and trained to
better cope with stress situations.

Lazarus & Folkman18
claim that when people make use of optimism to face adversity they are prevented
from thinking and acting to solve the problem because the purpose is only to
modify the accompanying emotional content and facilitate the adaptative process.

Pargament24
described the relevance of religious-spiritual coping when people turn to religion
to alleviate their problems. Tarakeshwar & Pargament31 described
the five main goals of religion: search of meaning, control, spiritual comfort,
closeness to God and to other social members, life transformation, and search
for physical, psychological, and emotional well-being.

Another coping
strategy is to search for information about the disease. It helps family to
recover a sense of self-help and control over the situation and open up a possibility
of helping the family member with an eating disorder. These mechanisms attenuate
suffering by lessening the feeling of impotence. However, Wiserman37
says that, in addition to searching for and acquiring information, it is paramount
to use this information to help the member's recovery by changing behaviors
and restructuring their daily life.

The intensity and
continuity of care can provide more knowledge on the disease and reduce anxiety
created by earlier lack of knowledge when the family was not able to help the
patient as intended.

Eating disorders
evoke strong feelings that are sometimes contradictory. Physical and social
aspects of family life are also affected by physical burnout and daily commitment,
precipitating crisis and disrupting the family dynamics. The disease potentiates
already existing conflicts and creates new ones, wearing out attachments and
inflicting psychic pain. In this scenario of changes and deadlocks, the family
structure has to be restructured so its members can face the new reality and
adjust to life conditions produced by disease and treatment.

The family is a
major component of treatment. Family dynamics can play a role in disease manifestation
or its persistence. Conflictive family relationships, mostly marked by no limit
setting, produce intense suffering. Overprotection, high expectations, and values
centered on success and external appearance are also part of this family dynamics.19,21,28
The body of the sick member concentrates these limitations, needs and difficulties,
and the symptom is an important form of expression and denunciation.

There should be
noted some limitations of the present study: 1 - constraints of the search (16-year
period, inclusion of studies in English, Spanish, French and Portuguese only),
leaving out other studies; 2- exclusion of non-indexed studies (theses and chapters
of books) may have resulted in few studies; 3- the majority of studies had design
flaws (sampling strategies and analysis techniques) that compromised their quality;
4- varied levels of relationship of informants in the studies; 5- varying disease
severity found in the different studies.

In conclusion,
besides parents, family members showed similar views about the disease affecting
another member of the family. Care provided to patients should include the opportunity
of examining and consulting family members at diagnosis and interventions. These
interventions can meet demands for clarifications and general information about
patient care (advice of psychoeducational nature is recommended) and situations
involving pathological functioning of patients and their family (complex interventions
for restructuring of personality and family dynamics are recommended). Support
networks with staff trained to diagnose and provide interventions should be
strongly considered and low-cost procedures may have positive effects on patient
management such as meeting with families experiencing similar situations or
self-help networks.